Solitary lingual neurofibroma. A rare case report
Eleonora BLIOUMI, Chrysoula TSOBANIDOU, Stefanos STEFANIDIS, Lampros ZOULOUMIS
Dental Department at “Agios Pavlos” Hospital of Thessaloniki (Head: Th. Floras), Dental Department at “Agios Dimitrios” Hospital of Thessaloniki (Head: S Stefanidis), Department of Histopathology, General Hospital “Agios Dimitrios” (Head: C. Tsobanidou), Department of Oral and Maxillofacial Surgery (Head: Prof. K. Antoniadis), Dental School, Aristotle University, Thessaloniki
Hellenic Archives of Oral & Maxillofacial Surgery (2016) 1, 27-33
SUMMARY: Neurofibroma is a rare benign, non-odontogenic tumour of the oral cavity. It consists of a wide variety of Schwann cells, perineural cells and fibroblasts. Clinically, neurofibromas may present as slow-growing, pedunculated or sessile tumours, which are usually asymptomatic. They may occur either as solitary lesions, or as individual manifestations of von Recklinghausen multiple neurofibromatosis.
In this study, we present the case of a patient with a solitary, asymptomatic, submucosal swelling of the lateral surface of the tongue. Surgical resection was the treat- ment of choice. In histopathological terms, this lesion was a neurofibroma. This was confirmed by its positive im- munoreactivity to S-100 protein, which is indicative of neural origin. The differential diagnosis and treatment of the lesion are discussed in detail, along with the clinical and laboratory findings.
What makes this case report especially interesting is exactly the rarity of oral neurofibromas.
KEY WORDS: Tongue, neurofibroma, S-100 protein
REFERENCES
Bharath TS, Krishna YR, Nalabolu GR, Pasupuleti S, Surapaneni S, Ganta SB: Neurofibroma of the palate. Hindawi Publishing Corporation, Case reports in Dentistry ID 898505, 2014
Deichler J, Martinez R, Niklander S, Seguel H, Marshall M, Esguep A: Solitary intraosseous neurofibroma of the mandible. A propos of a case. Med Oral Patol Oral Cir Bucal 1; 16 (6):e704-707, Sep 2011
Depprich R, Singh DD, Reinecke P, Kubler NR, Handschel J: Solitary submucous neurofibroma of the mandible: review of the literature and report of a rare case. Head & Face medicine 5:24, 2009
Go HJ: Benign peripheral nerve sheath tumor of the tongue. Yonsei Medical Journal 43:687-680, 2002
Gómez-Oliveira G, Fernández-Alba Luengo J, Martín-Sastre R, Patiño- Seijas B, López-Cedrún-Cembranos JL: Plexiform neurofibroma of the cheek mucosa. A case report. Med Oral 9:263-267, 2004
Iatrou I, Fotopoulos D, Theologie-Lygidakis N, Michail A: Preauricular facial nerve neurilemmoma in a paediatric patient. A case report. Hellenic Archives of Oral and Maxillofacial Surg 2, 113- 120, 2011
Ide F, Shimoyama T, Horie N, Kusama K: Comparative ultrastructural and immunohistochemical study of perineuroma and neurofi- broma of the oral mucosa. Oral Oncol 40:948-953, 2004
Jangam SS, Ingole SN, Deshpande MD, Ranadive PA: Solitary in- traosseous neurofibroma: report of a unique case. Contemp Clin Dent 5(4): 561–563, Oct-Dec 2014
Jham BJ, Costa NL, Batista AC, Mendonca EF: Traumatic neuroma of the mandible: a case report with spontaneous remission. J Clin Exp Dent 6(3):e317-202014, 2014
Maruyama M, Fushiki H, Watanabe Y: Solitary neurofibroma of the floor of the mouth: a case report. Hindawi Publishing Corporation, Case reports in Otolaryngology ID 967896, 2011
Misra S, Gogri P, Misra N, Bhandari A: Recurrent neurofibroma of the orbit. Australas Med J 6: 189–191, 2013
Neville BW, Damm DD, Allen CM, Bouquot JE: Oral & Maxillofacial pathology. 2nd Edition by W.B. Saunders Company 2002, pp.457- 458
Polak M, Polak G, Brocheriou C, Vigneul J: Solitary neurofibroma of the mandible: case report and review of the literature. J Oral Max- illofac Surg 47:65-68, 1989
Ramakrishnan P, Sylvester V, Screenivasan P, Vengalath J, Valambath S: Hypertrichotic giant nevus spilus tardivus and neurofibroma of the tongue in sporadic von Recklinghausen’s disease. Hindawi Publishing Corporation. Case Reports in Dermatological Medicine ID 141075, 2014
Sahni P, Singhvi AS, Nayak MT: Solitary neurofibroma of tongue: a rare case report. Dental Impact 4(1): June, 2012
Salla JT, Johann AC, Carcia BG, Aguiar MC, Mesquita RA: Retrospective analysis of oral peripheral nerve sheath tumors in Brazilians. Braz Oral Res 23(1): 43-48, 2009
Sheejith M, Joseph B, Nath SG, Sheejith B: An unusual case of multiple intraoral manifestations of neurofibromatosis type 1: case report with literature review. Clin Diagn Res 8(12): ZD20–ZD22, Dec 2014
Shekar V, Rangdol V, Baliah WJ, Thirunavukarasu S: An unusual oral manifestation of type 1 neurofibromatosis: A case report and a review of literature. J Nat Sci Biol Med 6(1): 261–263, Jan-Jun 2015
Sivapathasundharam B, Lavanya S, Deepalakshmi M, Saravanakumar R, Ahathya RS: Solitary neurofibroma of the gingiva. J Oral Maxillofac Pathol 8: 107–109, 2004
Αγγελόπουλος ΑΠ, Παπανικολάου Σ, Αγγελοπούλου Ε. Σύγχρονη στοματική και γναθοπροσωπική παθολογία. 3η Έκδοση. Ιατρικές Εκδόσεις Λίτσας 2000, σελ. 412-414
Dental Department at “Agios Pavlos” Hospital of Thessaloniki (Head: Th. Floras), Dental Department at “Agios Dimitrios” Hospital of Thessaloniki (Head: S Stefanidis), Department of Histopathology, General Hospital “Agios Dimitrios” (Head: C. Tsobanidou), Department of Oral and Maxillofacial Surgery (Head: Prof. K. Antoniadis), Dental School, Aristotle University, Thessaloniki
Hellenic Archives of Oral & Maxillofacial Surgery (2016) 1, 27-33
SUMMARY: Neurofibroma is a rare benign, non-odontogenic tumour of the oral cavity. It consists of a wide variety of Schwann cells, perineural cells and fibroblasts. Clinically, neurofibromas may present as slow-growing, pedunculated or sessile tumours, which are usually asymptomatic. They may occur either as solitary lesions, or as individual manifestations of von Recklinghausen multiple neurofibromatosis.
In this study, we present the case of a patient with a solitary, asymptomatic, submucosal swelling of the lateral surface of the tongue. Surgical resection was the treat- ment of choice. In histopathological terms, this lesion was a neurofibroma. This was confirmed by its positive im- munoreactivity to S-100 protein, which is indicative of neural origin. The differential diagnosis and treatment of the lesion are discussed in detail, along with the clinical and laboratory findings.
What makes this case report especially interesting is exactly the rarity of oral neurofibromas.
KEY WORDS: Tongue, neurofibroma, S-100 protein
REFERENCES
Bharath TS, Krishna YR, Nalabolu GR, Pasupuleti S, Surapaneni S, Ganta SB: Neurofibroma of the palate. Hindawi Publishing Corporation, Case reports in Dentistry ID 898505, 2014
Deichler J, Martinez R, Niklander S, Seguel H, Marshall M, Esguep A: Solitary intraosseous neurofibroma of the mandible. A propos of a case. Med Oral Patol Oral Cir Bucal 1; 16 (6):e704-707, Sep 2011
Depprich R, Singh DD, Reinecke P, Kubler NR, Handschel J: Solitary submucous neurofibroma of the mandible: review of the literature and report of a rare case. Head & Face medicine 5:24, 2009
Go HJ: Benign peripheral nerve sheath tumor of the tongue. Yonsei Medical Journal 43:687-680, 2002
Gómez-Oliveira G, Fernández-Alba Luengo J, Martín-Sastre R, Patiño- Seijas B, López-Cedrún-Cembranos JL: Plexiform neurofibroma of the cheek mucosa. A case report. Med Oral 9:263-267, 2004
Iatrou I, Fotopoulos D, Theologie-Lygidakis N, Michail A: Preauricular facial nerve neurilemmoma in a paediatric patient. A case report. Hellenic Archives of Oral and Maxillofacial Surg 2, 113- 120, 2011
Ide F, Shimoyama T, Horie N, Kusama K: Comparative ultrastructural and immunohistochemical study of perineuroma and neurofi- broma of the oral mucosa. Oral Oncol 40:948-953, 2004
Jangam SS, Ingole SN, Deshpande MD, Ranadive PA: Solitary in- traosseous neurofibroma: report of a unique case. Contemp Clin Dent 5(4): 561–563, Oct-Dec 2014
Jham BJ, Costa NL, Batista AC, Mendonca EF: Traumatic neuroma of the mandible: a case report with spontaneous remission. J Clin Exp Dent 6(3):e317-202014, 2014
Maruyama M, Fushiki H, Watanabe Y: Solitary neurofibroma of the floor of the mouth: a case report. Hindawi Publishing Corporation, Case reports in Otolaryngology ID 967896, 2011
Misra S, Gogri P, Misra N, Bhandari A: Recurrent neurofibroma of the orbit. Australas Med J 6: 189–191, 2013
Neville BW, Damm DD, Allen CM, Bouquot JE: Oral & Maxillofacial pathology. 2nd Edition by W.B. Saunders Company 2002, pp.457- 458
Polak M, Polak G, Brocheriou C, Vigneul J: Solitary neurofibroma of the mandible: case report and review of the literature. J Oral Max- illofac Surg 47:65-68, 1989
Ramakrishnan P, Sylvester V, Screenivasan P, Vengalath J, Valambath S: Hypertrichotic giant nevus spilus tardivus and neurofibroma of the tongue in sporadic von Recklinghausen’s disease. Hindawi Publishing Corporation. Case Reports in Dermatological Medicine ID 141075, 2014
Sahni P, Singhvi AS, Nayak MT: Solitary neurofibroma of tongue: a rare case report. Dental Impact 4(1): June, 2012
Salla JT, Johann AC, Carcia BG, Aguiar MC, Mesquita RA: Retrospective analysis of oral peripheral nerve sheath tumors in Brazilians. Braz Oral Res 23(1): 43-48, 2009
Sheejith M, Joseph B, Nath SG, Sheejith B: An unusual case of multiple intraoral manifestations of neurofibromatosis type 1: case report with literature review. Clin Diagn Res 8(12): ZD20–ZD22, Dec 2014
Shekar V, Rangdol V, Baliah WJ, Thirunavukarasu S: An unusual oral manifestation of type 1 neurofibromatosis: A case report and a review of literature. J Nat Sci Biol Med 6(1): 261–263, Jan-Jun 2015
Sivapathasundharam B, Lavanya S, Deepalakshmi M, Saravanakumar R, Ahathya RS: Solitary neurofibroma of the gingiva. J Oral Maxillofac Pathol 8: 107–109, 2004
Αγγελόπουλος ΑΠ, Παπανικολάου Σ, Αγγελοπούλου Ε. Σύγχρονη στοματική και γναθοπροσωπική παθολογία. 3η Έκδοση. Ιατρικές Εκδόσεις Λίτσας 2000, σελ. 412-414
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